(2006) 44, 509–513 & 2006 International Spinal Cord Society All rights reserved 1362-4393/06 $30.00 www.nature.com/sc

Original Article

Cervical : a seemingly still neglected symptom of cervical spine disorder?

H Nakajima*,1, K Uchida1, S Kobayashi1, Y Kokubo1, T Yayama1, R Sato1, T Inukai1, T Godfrey1 and H Baba1 1Division of Orthopaedics and Rehabilitation , Department of , School of Medicine, University of Fukui, Fukui, Japan

Design: A review of 10 surgical cases with symptoms of cervical angina. Objective: To stress the importance of symptoms of cervical angina in patients with cervical spine disorders. Setting: Fukui University Hospital, Japan. Results: A total of 10 patients complaining of symptoms of cervical angina were admitted with a tentative diagnosis of coronary disease. Pain relief was achieved by anterior surgical decompression in all patients. Conclusion: We stress that should be aware of the symptoms of cervical angina and that surgical intervention often leads to complete relief of symptoms. Spinal Cord (2006) 44, 509–513. doi:10.1038/sj.sc.3101888; published online 6 December 2005

Keywords: cervical angina; cervical spondylosis; ossification of the posterior longitudinal ; surgery

Introduction Patients and methods Cervical spine disorders may often be present with pain Between 1991 and 2004, a total of 706 patients under- in the upper anterior chest and scapular areas, went cervical spine because of neurological resembling true angina pectoris.1–3 Anterior chest pain symptoms and signs, such as (n ¼ 314), associated with cervical intervertebral disc diseases, myeloradiculopathy (n ¼ 162), radiculopathy (n ¼ 198), ossified posterior longitudinal ligament (OPLL), or or so-called ‘discopathy’ (n ¼ 32). Reviewing the clinical other spinal disorders, has been sometimes described charts in retrospect, 10 patients had presented with 3,4 as ‘cervical angina’ and appears to be relatively cervical angina as the main symptom. Of these, three unknown clinical syndrome. Prompt and accurate patients visited our hospital directly when the neck and diagnosis requires a strong sense of suspicion in patients 5 anterior chest pain symptoms appeared, but the clinical with inadequately explained chest pain. Jacobs sug- presentations of the other seven cases were considered gested that C6 and C7 roots are the most different and included true angina pectoris and neurosis. frequently involved and the pain is possibly mediated These 10 cases were retrospectively reviewed with via the medial and lateral pectoral . Spine respect to the clinical features, radiological findings, specialists should be well aware of this presentation and the results of other specific tests. in their routine clinical examinations but, unfortunately Radiographic examination included flexion-extension and in fact, a number of patients still appear to be lateral films and, in some cases, discography. In our diagnosed as coronary artery disease, and thus undergo Neuro-orthopaedic Unit, myelography was excluded unnecessary examinations and medications. from routine radiological examination since 1996 due In the present short communication, we describe 10 to its biological invasiveness. Radiological examination surgical cases in whom cervical spine disorders were included the following workup as we reported pre- misdiagnosed over long periods. We emphasize the 6,7 importance of these clinical symptoms in the diagnosis viously. (i) Lordotic alignment of the cervical spine on of cervical spine disorders. radiographs taken in neutral positions, designated as cervical spine lordosis. (ii) Reduction of bony spinal canal size, measured on lateral films, at suspected *Correspondence: H Nakajima, Division of Orthopaedics and vertebral level responsible for neural compression. Rehabilitation Medicine, Department of Surgery, School of Medicine, University of Fukui, Shimoaizuki 23, Matsuoka, Fukui 910-1193, (iii) Lordotic alignment of the cervical spinal cord on Japan magnetic resonance imaging (MRI; 1.5 Tesla Signa, Cervical angina H Nakajima et al 510

General Electric, Milwaukee, WI, USA), termed spinal cord lordosis. Other radiographic abnormalities were intervertebral disc space narrowing, spondylotic osteo-

phyte formation, and the existence of OPLL. In (years) Follow-up addition, MR angiography was conducted in three patients who suffered from other symptoms, such as vertebral insufficiency syndrome. All surgeries were performed by one of the authors ompression (Robinson’s Pain relief

(H Baba) using a uniform surgical technique as described post-op (m) 6–9

previously. A left-side anterolateral oblique incision a: difficulty of breathing; b: was pursued for the anterior cervical spine followed by Robinson’s anterior decompression and interbody fusion or subtotal spondylectomy with autologous iliac grafting. In OPLL, the essential technique was resection

of the ossified plaque anteriorly with complete decom- )) 0.1) 4.5 0.1 0.5 2.0 2.0

9 À À À pression of the spinal cord. Neurological assessment Other medical examinations after surgery was performed by independent observers other than the principal surgeon. A written informed consent was obtained from all patients and the study strictly followed the Guidelines of the Ethical Committee of Fukui University. Other symptoms

Results

Clinical presentation before and after surgical treatment of pain Localization Table 1 shows the preoperative and postoperative clinical demographic data of the 10 patients (six men and four women). The average age of these patients was 54.5 years (range: 36–74 years). The average duration of symptoms prior to definitive diagnosis was 5.6 months (range: 2–12 months). The background disease was cervical spondylotic myelopathy (CSM, n ¼ 3), cervical disc herniation (CDH, n ¼ 6), and OPLL Myelopathy/ radiculopathy (n ¼ 1). The affected levels were C4–5 level in three cases, C5–6 level in four cases, and C6–7 level in three cases. All patients improved after anterior decompressive Pre/post surgeries. JOA score We classified the chest pain based on its localization. Five cases had retrosternal pain, three cases had left lower anterior chest pain, and two cases had epigastric pain. Five cases had autonomic symptoms (eg, difficulty of breathing, vertigo, and headache). Upper arm Surgical procedure neurological symptoms and left lower anterior chest pain tended to appear simultaneously in patients with

radiculopathy, and autonomic symptoms tended to levels appear conspicuously rather than upper arm symptoms Affected in the myelopathy cases.

Illustrative case presentation Case 6: A 54-year-old woman was admitted to our University Hospital with complaints of left upper arm

pain and left lower anterior chest pain especially when (m) Disease symptoms

putting her right arm down. She had undergone Duration of angiography at other hospitals for suspected cardiovas- cular disease or thoracic outlet syndrome. The duration Perioperative clinical data of the 10 patients sex of symptoms prior to definitive diagnosis was 3 months. Age MRI findings were presence of cervical disc herniation (years) at right-sided C5–6 level (Figure 1). A diagnosis of

cervical angina was made, and she underwent anterior Table 1 Case No. 123 694 (F) 37 (M)5 69 (M)6 48 6.0 (M)7 3.0 67 (M)8 8.0 CSM 549 (F) 12.0 CDH 36 (M)10 CSM 3.0 74 CDH (M) C5–C6CSM: 54 3.0 cervical (F) C4–C5 12.0 spondylotic 37 CSM myelopathy; (F) C6 CDH:procedure); cervical Subt Subt: C4–C5 disc subtotal C5 herniation; spondylectomy; 2.0 Subt C6–C7 CDH OPLL:vertigo; JOA: OPLL ossification c: Japanese of occipital Orthopaedics C5 the headache; Association; posterior Subt 2.0 C6–7 longitudinal UCG: 1: C4–C5 Ant ligament; ultrasonic retrosternal; CDH 5.0 Ant: 2: cardiography, anterior left BR: dec lower 11/15 brain C6–C7 anterior C5–C6 C4, MRI, CDH chest; 5 12/14 PSY: 3: Subt consultation CDH epigastric; with C6–7 C5–6 Myelopathy a 13/16 10/14 Ant Ant psychiatrist, C5–C6 AG: Radiculopathy angiography 15/17 Radiculopathy C5–6 Myelopathy C6–C7 Ant C5–C6 15/17 15/17 C6–7 Myelopathy Ant C5–6 Ant Myelopathy Radiculopathy 5/7 2 1 15/17 1 16/17 Myelopathy 3 Radiculopathy Myelopathy a 1 2 1 a 2 3 a, (+)/UCG, ( b, BR c 1 (+)/PSY (+)/UCG a, c (+)/UCG, (+)/AG BR b, c (+)/AG 3.0 (+)/PSY, BR 0.5 0.2 ( ( 3.0 4.8 1.0 0.1 4.0 2.5 3.5 3.8 3.4 3.0 2.2

Spinal Cord Cervical angina H Nakajima et al 511 decompression followed by interbody fusion (Robin- months elapsed before making a definitive diagnosis. son’s procedure) at C5–6 level. Pain-related symptoms X-ray findings included presence of segmental OPLL including chest pain improved immediately after at C5 and C6. MRI findings included presence of surgery. segmental OPLL and cervical disc herniation on left-side Case 7: A 36-year-old man was admitted to our of C6–7 (Figure 2). Diagnosis of cervical angina was University Hospital with complaints of left upper arm made, and he underwent anterior decompression with pain and difficulty of breathing, vertigo, and headache. interbody fusion at C5–7 level (C6 subtotal spondylect- Neurological examination revealed a brisk deep omy). The autonomic symptoms improved immediately reflex of upper and lower limbs. He had consulted after surgery while the other myelopathy symptoms a cardiovascular and a neurosurgeon, and 12 improved gradually.

Discussion Among the multitude of symptoms of cervical spine disorders, cervical angina may be miscellaneous, but it must be always recognized in clinical practice.4,10–12 In addition, the symptoms tend to be misidentified more frequently in elderly individuals because of increased incidence of coronary artery diseases. The symptom is rather easily recognizable when the patient presents with neurological signs of spinal cord compromise, however, actually frequently, it appears to be a missing problem without careful examination. Many investigators1–5,10–12 have described details of this status but it appears still neglected in the routine clinical practice. Oille13 was perhaps the first to describe the symptom in 1937 patients with chest pain of cervical nerve root origin. Jacobs5 also reported a large series of 164 cases 14 Figure 1 MRI (TR, 4000 ms; TE, 80 ms) showed presence of with cervical angina observed over 20 years. Brodsky cervical disc herniation on the right-side at C5–6 level (arrow). reported the largest series of 438 cases with cervical (a) sagittal view, (b) axial view angina, and perhaps had made the finest insight into the

Figure 2 (a) Lateral plain radiograph showed the presence of segmental OPLL at C5 and C6. MRI (TR, 4000 ms; TE, 80 ms) showed presence of segmental OPLL (arrowhead) and cervical disc herniation on the left-side at C6–7 level (arrow). (b) sagittal view, (c) axial view

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physiological etiologies. On the other hand, Iwasa15 were more evident than upper arm symptoms. There- found only three (5%) of 63 patients with angina fore, cervical angina may be more difficult to diagnose pectoris had cervical nerve root compression, and and there is a need for great care especially in LaBan et al12 also described a low incidence of true myelopathy cases. cervical angina. Why there is a wide spectrum of Routine MRI examination, or even if myelopathy is incidence of cervical angina? The frequency may be a suspected, is insufficiently informative for the functional continuing quest mainly because of differences in patient assessment of cervical angina. Perhaps, discography sampling, diagnostic criteria, and more importantly, and/or selective nerve root infiltration with xylocaine variability of symptoms. Reviewing 706 patients who block may be the best tool to make a functional underwent cervical spine surgeries between 1991 and diagnosis of cervical angina syndrome associated with 2004, we found approximately 1.4% of those were spinal cord and/or nerve root compression. However, considered to have exhibited symptoms of cervical these invasive tests should be considered carefully. On angina. However, we presume the true frequency to be the other hand, F-2-fluoro-deoxy-D-glucose positron actually higher because there were cases examiners or emission tomography may be useful for detection of patients did not recognize these symptoms as cervical neural dysfunction of the spinal cord17 affecting the spine disorders. somatic as well as autonomic nervous disorders around There is a debate on the physiological etiology of the chest. Rest and neck collar fixation or nitro-glycerine cervical angina syndrome. Brodsky15 indicated that medication may be recommended as an alternative radicular pain is the direct cause, and LaBan et al12 approach to establish the diagnosis. It is obvious that presumed that the spinal cord ventral roots produced defective coronary artery circulation is an extremely protopathic pain around the chest. Gonza´ lez-Darder serious problem. Once coronary artery disease has been et al16 considered deactivation of the descending adequately excluded, the possibility of a cervical angina inhibitory system as the main cause of symptoms. Other syndrome should be considered, especially if accompa- possible factors may be sympathetic pain and sino- nied by signs of cervical radiculopathy or myelopathy. vertebral pain. When it is difficult to distinguish between true angina Although coronary arteriography has advanced the pectoris and cervical angina, adequate coronary diag- diagnosis of angina pectoris and distinction between nostic studies are imperative. chest pain of coronary origin as opposed to that arising A careful approach should be followed when treating from other structures, unnecessary invasive procedures cervical angina syndrome. Several groups3,5,14 have should be avoided, if possible. Prompt and accurate observed spontaneous resolution of the symptoms or diagnosis requires a strong sense of suspicion in patients that a simple external neck fixation helps in pain with inadequately explained chest pain. According to elimination, either transiently or permanently. Approxi- the Jacobs’s observation,5 common manifestations mately three quarters of the patients had been estimated associated with cervical angina include neck and arm to improve with conservative treatment.14 However, in pain, upper arm radicular symptoms and fatigue, cases where neurological compromise is evident by parasternal tenderness, and occipital headache. spinal cord and/or nerve root compression, surgery may Brodsky14 stressed the presence of associated autonomic be necessary to produce rapid improvement. Patients or other symptoms such as dyspnea, nausea, diaphor- with cervical angina suffer from discomfort and uneasy esis, diplopia, and sympathetic nervous signs. These chest pain. When these symptoms continue and the complex symptoms appear well known when the patient does not show response to conservative , patients have compression of the cervical spinal cord one must consider surgery, since, at least occasionally, and/or cervical nerve root. Constant1 indicated that an aimless conservative therapy is not good. We believe chest pain is likely nonanginal if its duration is 430 min that unnecessary or doubtful surgical intervention must or o5 s, increases with inspiration, can be induced be strictly avoided. However, based on our experience by a single movement of the trunk or arm, by local even in a small series, anterior cervical surgery to correct fingers pressure or bending forward, or if it disappears nerve root or spinal cord compression can be a useful immediately on lying down. There are also many option for pain relief. On the other hand, regarding presumptive signs of nonanginal chest pain such as cardiac examination, physicians must always be aware localization with one finger, radiation to the nuchal of this miscellaneous syndrome in order to avoid area, an inflammatory primary site, pain that unnecessary coronary artery examination. In this point reaches maximum at onset, or relief within a few of view, appropriate and cooperative approach in seconds of swallowing food. Neurological sign and physical examination must be followed among the oesophageal spasm are features that help rule out specialists. angina. However, the possibility of coexistent organic coronary disease and cervical angina must be kept in mind. Acknowledgements In our cases, left lower chest pain tended to appear as This work was supported in part by a grant (2003) from the a radicular sign, retrosternal, epigastric pain, and Investigation Committee on Ossification of the Spinal Liga- autonomic symptoms tended to be accompany myelo- ments, the Bureau of the Japanese Ministry of pathy. In the myelopathy cases, autonomic symptoms Health and Welfare.

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